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Open Access 01-12-2021 | Polysomnography | Research

Sleep problems among children with Fetal Alcohol Spectrum Disorders (FASD)- an explorative study

Authors: Katarzyna Anna Dylag, Bożena Bando, Zbigniew Baran, Paulina Dumnicka, Katarzyna Kowalska, Paulina Kulaga, Katarzyna Przybyszewska, Jakub Radlinski, Sylvia Roozen, Leopold Curfs

Published in: Italian Journal of Pediatrics | Issue 1/2021

Abstract

Background

Fetal alcohol spectrum disorders (FASD) is a group of conditions resulting from prenatal alcohol exposure (PAE). Patients with FASD experience a variety of neuropsychological symptoms resulting from central nervous system impairment. Little is known about sleep disorders associated with PAE. The objective of this study was to investigate sleep problems related to FASD.

Methods

Forty patients (median age 8 years (6; 11)) diagnosed with FASD and forty typically developing children (median age 10 years (8; 13)) were recruited for the 1st phase of the study. In the 1st phase, the screening of sleep problems was performed with Child Sleep Habit Questionnaire (CSHQ) filled in by a caregiver. Those of the FASD group who scored above 41 points were qualified to the 2nd phase of the study and had an in-lab attended polysomnography (PSG) performed. The measurements consisted of electroencephalogram, electrooculograms, chin and tibial electromyogram, electrocardiogram, ventilatory monitoring, breathing effort, pulse oximetry, snoring and body position. Their results were compared to PSG laboratory reference data.

Results

The number of participants with sleep disturbances was markedly higher in the FASD group as compared to typically developing children (55% vs. 20%). The age-adjusted odds ratio for a positive result in CSHQ was 4.31 (95% CI: 1.54–12.11; p = 0.005) for FASD patients as compared to the control group. Significant differences between the FASD as compared to the typically developing children were observed in the following subscales: sleep onset delay, night wakings, parasomnias, sleep disordered breathing, and daytime sleepiness. Children from the FASD group who underwent PSG experienced more arousals during the sleep as compared with the PSG laboratory reference data. The respiratory indices in FASD group appear higher than previously published data from typically developing children.

Conclusion

The results support the clinical observation that sleep disorders appear to be an important health problem in individuals with FASD. In particular distorted sleep architecture and apneic/hypopneic events need further attention.

Hoyme HE, Kalberg WO, Elliott AJ, Blankenship J, Buckley D, Marais A-S, et al. Updated clinical guidelines for diagnosing fetal alcohol spectrum disorders. Pediatrics. 2016;138(2).

Roozen S, Peters G-JY, Kok G, Townend D, Nijhuis J, Curfs L. Worldwide prevalence of fetal alcohol spectrum disorders: a systematic literature review including meta-analysis. Alcohol Clin Exp Res. 2016;40(1):18–32 Available from: http://doi.wiley.com/10.1111/acer.12939.PubMedCrossRef

Popova S, Lange S, Probst C, Gmel G, Rehm J. Estimation of national, regional, and global prevalence of alcohol use during pregnancy and fetal alcohol syndrome: a systematic review and meta-analysis. Lancet Glob Health. 2017;5(3):e290–9 Available from: www.thelancet.com/lancetgh. Cited 2021 Feb 16.PubMedCrossRef

Ehrhart F, Roozen S, Verbeek J, Koek G, Kok G, van Kranen H, et al. Review and gap analysis: molecular pathways leading to fetal alcohol spectrum disorders. Mol Psychiatry. 2019;24:10–7 Available from: https://doi.org/10.1038/s41380-018-0095-4. Nature Publishing Group. Cited 2020 Nov 4.PubMedCrossRef

Mattson SN, Bernes GA, Doyle LR. Fetal alcohol spectrum disorders: a review of the neurobehavioral deficits associated with prenatal alcohol exposure. Alcohol Clin Exp Res. 2019;43:1046–62 Available from: /pmc/articles/PMC6551289/?report=abstract. Blackwell Publishing Ltd. Cited 2020 Sep 16.PubMedPubMedCentral

McDougall S, Finlay-Jones A, Arney F, Gordon A. A qualitative examination of the cognitive and behavioural challenges experienced by children with fetal alcohol spectrum disorder. Res Dev Disabil. 2020;104 Available from: https://pubmed-1ncbi-1nlm-1nih-1gov-1nb5yeiky0096.hanproxy.cm-uj.krakow.pl/32521433/. Cited 2021 Feb 11.

Kingdon D, Cardoso C, JJ MG. Research Review: Executive function deficits in fetal alcohol spectrum disorders and attention-deficit/hyperactivity disorder – a meta-analysis. J Child Psychol Psychiatry Allied Discip. 2016;57(2):116–31 Available from: https://pubmed-1ncbi-1nlm-1nih-1gov-1nb5yeiky0096.hanproxy.cmuj.krakow.pl/26251262/. Cited 2021 Feb 11.CrossRef

Kully-Martens K, Denys K, Treit S, Tamana S, Rasmussen C. A review of social skills deficits in individuals with fetal alcohol spectrum disorders and prenatal alcohol exposure: profiles, mechanisms, and interventions. Alcohol Clin Exp Res. 2012;36:568–76 Available from: https://pubmed.ncbi.nlm.nih.gov/22017360/. Cited 2021 Feb 12.PubMedCrossRef

Roozen S, Olivier L, Niemczyk J, von Gontard A, Peters G-JY, Kok G, et al. Nocturnal incontinence in children with fetal alcohol spectrum disorders (FASD) in a South African cohort. J Pediatr Urol. 2017; Available from: http://www.ncbi.nlm.nih.gov/pubmed/28381366. Cited 2017 Apr 15

10.

Roozen S, Dylag KA, Przybyszewska K, Niemczyk J, von Gontard A, Peters GJY, et al. Incontinence in persons with fetal alcohol spectrum disorders: a polish cohort. J Pediatr Urol. 2020;16(3):386.e1–386.e11 Available from: https://pubmed.ncbi.nlm.nih.gov/32222270/. Cited 2021 Feb 13.CrossRef

11.

Amos-Kroohs RM, Fink BA, Smith CJ, Chin L, Van Calcar SC, Wozniak JR, et al. Abnormal eating behaviors are common in children with fetal alcohol spectrum disorder. J Pediatr. 2016;169:194–200.e1 Available from: http://www.ncbi.nlm.nih.gov/pubmed/26608087. Cited 2019 Aug 29.PubMedCrossRef

12.

Werts RL, Van Calcar SC, Wargowski DS, Smith SM. Inappropriate feeding behaviors and dietary intakes in children with fetal alcohol spectrum disorder or probable prenatal alcohol exposure. Alcohol Clin Exp Res. 2014;38(3):871–8 Available from: http://www.ncbi.nlm.nih.gov/pubmed/24164456. Cited 2017 Apr 12.PubMedCrossRef

13.

Kamara D, Beauchaine TP. A review of sleep disturbances among infants and children with neurodevelopmental disorders. Rev J Autism Dev Disord. 2020;7:278–94 Available from: https://pubmed-1ncbi-1nlm-1nih-1gov-1nb5yeiti0033.hanproxy.cm-uj.krakow.pl/33344102/. Springer. Cited 2021 Feb 6.PubMedCrossRef

14.

DelRosso LM, Picchietti DL, Spruyt K, Bruni O, Garcia-Borreguero D, Kotagal S, et al. Restless sleep in children: a systematic review. Sleep Med Rev. 2021;56:101406 W.B. Saunders Ltd.PubMedCrossRef

15.

Shelton AR, Malow B. Neurodevelopmental disorders commonly presenting with sleep disturbances. Neurotherapeutics. 2021 Available from: https://pubmed.ncbi.nlm.nih.gov/33403472/. Springer Science and Business Media Deutschland GmbH. Cited 2021 Feb 12

16.

Hanlon-Dearman A, Chen ML, Olson HC. Understanding and managing sleep disruption in children with fetal alcohol spectrum disorder. Biochem Cell Biol. 2018;96:267–74 Available from: https://pubmed-1ncbi-1nlm-1nih-1gov-1nb5yeivc0039.hanproxy.cm-uj.krakow.pl/28977761/. Canadian Science Publishing. Cited 2021 Feb 12.PubMedCrossRef

17.

Ipsiroglu OS, Wind K, Hung YH (Amy), Berger M, Chan F, Yu W, et al. Prenatal alcohol exposure and sleep-wake behaviors: exploratory and naturalistic observations in the clinical setting and in an animal model. Sleep Med. 2019;54:101–12 Available from: https://pubmed-1ncbi-1nlm-1nih-1gov-1nb5yeivc0011.hanproxy.cmuj.krakow.pl/30530254/. Cited 2021 Feb 12.PubMedCrossRef

18.

Mughal R, Hill CM, Joyce A, Dimitriou D. Sleep and cognition in children with fetal alcohol spectrum disorders (FASD) and children with autism spectrum disorders (ASD). Available from: www.mdpi.com/journal/brainsci. Cited 2021 Feb 11

19.

Mughal R, Joyce A, Hill C, Dimitriou D. Sleep disturbance as a predictor of anxiety in children with fetal alcohol spectrum disorders and typically developing children. Res Dev Disabil. 2020;101 Available from: https://pubmed-1ncbi-1nlm-1nih-1gov-1nb5yeivc0011.hanproxy.cm-uj.krakow.pl/32224364/. Cited 2021 Feb 12.

20.

Steinhausen HC, Spohr HL. Long-term outcome of children with fetal alcohol syndrome: Psychopathology, behavior, and intelligence. In: Alcoholism: Clinical and Experimental Research: Wiley; 1998. p. 334–8. Available from: https://onlinelibrary.wiley.com/doi/full/10.1111/j.1530-0277.1998.tb03657.x. Cited 2020 Sep 26.

21.

Ipsiroglu OS, Mckellin WH, Carey N, Loock C. Social science & medicine “They silently live in terror”. Why sleep problems and night-time related quality-of-life are missed in children with a fetal alcohol spectrum disorder. Soc Sci Med. 2013;79:76–83. https://doi.org/10.1016/j.socscimed.2012.10.027.CrossRefPubMed

22.

Inkelis SM, Thomas JD. Sleep in infants and children with prenatal alcohol exposure. Alcohol Clin Exp Res. 2018;42:1390–405 Available from: /pmc/articles/PMC6274610/. Blackwell Publishing Ltd. Cited 2021 Feb 13.CrossRef

23.

Yates JF, Troester MM, Ingram DG. Sleep in children with congenital malformations of the central nervous system. Curr Neurol Neurosci Rep. 2018;18 Available from: https://pubmed-1ncbi-1nlm-1nih-1gov-1nb5yeivc0039.hanproxy.cm-uj.krakow.pl/29789951/. Current Medicine Group LLC 1. Cited 2021 Feb 12.

24.

Hayes N, Moritz KM, Reid N. Parent-reported sleep problems in school-aged children with fetal alcohol spectrum disorder: association with child behaviour, caregiver, and family functioning. Sleep Med. 2020;74:307–314. Available from: https://pubmed-1ncbi-1nlm-1nih-1gov-1nb5yeivc0011.hanproxy.cm-uj.krakow.pl/32889299/. Cited 2021 Feb 12

25.

Chandler-Mather N, Occhipinti S, Donovan C, Shelton D, Dawe S. An investigation of the link between prenatal alcohol exposure and sleep problems across childhood. Drug Alcohol Depend. 2021;218:108412.PubMedCrossRef

26.

Pesonen AK, Räikkönen K, Matthews K, Heinonen K, Paavonen JE, Lahti J, et al. Prenatal origins of poor sleep in children. Sleep. 2009;32(8):1086–92 Available from: https://pubmed-1ncbi-1nlm-1nih-1gov-1nb5yeivc0011.hanproxy.cmuj.krakow.pl/19725260/. Cited 2021 Feb 12.PubMedPubMedCentralCrossRef

27.

Wengel T, Hanlon-Dearman AC, Fjeldsted B. Sleep and sensory characteristics in young children with fetal alcohol spectrum disorder. J Dev Behav Pediatr. 2011;32(5):384–92.PubMedCrossRef

28.

Goril S, Zalai D, Scott L, Shapiro CM. Sleep and melatonin secretion abnormalities in children and adolescents with fetal alcohol spectrum disorders. Sleep Med. 2016;23:59–64. https://doi.org/10.1016/j.sleep.2016.06.002.CrossRefPubMed

29.

Chen ML, Olson HC, Picciano JF, Starr JR, Owens J. Sleep problems in children with fetal alcohol spectrum disorders. J Clin Sleep Med. 2012;8(4):421–9 Available from: http://www.ncbi.nlm.nih.gov/pubmed/22893773. Cited 2019 Aug 29.PubMedPubMedCentralCrossRef

30.

Owens JA, Spirito A, McGuinn M. The children's sleep habits questionnaire (CSHQ): psychometric properties of a survey instrument for school-aged children. Sleep. 2000;23(8):1043–51.PubMedCrossRef

31.

Lewandowski AS, Toliver-Sokol M, Palermo TM. Evidence-Based Review of Subjective Pediatric Sleep Measures. J Pediatr Psychol. 2011;36(7):780–93 Available from: https://academic.oup.com/jpepsy/article-lookup/doi/10.1093/jpepsy/jsq119.PubMedPubMedCentralCrossRef

32.

Ophoff D, Slaats MA, Boudewyns A, Glazemakers I, Van Hoorenbeeck K, Verhulst SL. Sleep disorders during childhood: a practical review. Eur J Pediatr. 2018;177:641–8 Available from: https://pubmed.ncbi.nlm.nih.gov/29502303/.PubMedCrossRef

33.

Sen T, Spruyt K. Pediatric sleep tools: An updated literature review. Front Psychiatry. 2020;11:317 Available from: www.frontiersin.org.PubMedPubMedCentralCrossRef

34.

Berry RB, Brooks R, Gamaldo CE, Harding SM, Marcus CLCL, Vaughn BV, et al. The AASM manual for the scoring of sleep and associated events: rules, terminology and technical specifications, version 2.5. Darien: American Academy of Sleep Medicine; 2018.

35.

R Core Team. R: a language and environment for statistical computing. Vienna: R Foundation for Statistical Computing; 2019. Available from: https://www.r-project.org/

36.

Montgomery-Downs HE, O’Brien LM, Gulliver TE, Gozal D. Polysomnographic characteristics in normal preschool and early school-aged children. Pediatrics. 2006;117(3):741–53 Available from: http://www.ncbi.nlm.nih.gov/pubmed/16510654.PubMedCrossRef

37.

Goodwin JL, Kaemingk KL, Mulvaney SA, Morgan WJ, Quan SF. Clinical screening of school children for polysomnography to detect sleep-disordered breathing--the Tucson Children’s assessment of Sleep apnea study (TuCASA). J Clin Sleep Med. 2005;1(3):247–54 https://doi.org/10.5664/jcsm.26338.PubMedPubMedCentralCrossRef

38.

Owens JA, Spirito A, McGuinn M, Nobile C. Sleep habits and sleep disturbance in elementary school-aged children. J Dev Behav Pediatr. 2000;21(1):27–36 Available from: https://pubmed.ncbi.nlm.nih.gov/10706346/.PubMedCrossRef

39.

Alvik A, Mari A, Aalen OO, Lindemann R. Early human development binge alcohol exposure once a week in early pregnancy predicts temperament and sleeping problems in the infant. Early Hum Dev. 2011;87(12):827–833. Available from: https://doi.org/10.1016/j.earlhumdev.2011.06.009.

40.

Troese M, Fukumizu M, Sallinen BJ, Gilles AA, Wellman JD, Paul JA, et al. Sleep fragmentation and evidence for sleep debt in alcohol-exposed infants. Early Hum Dev. 2008;84(9):577–85 https://doi.org/10.1016/j.earlhumdev.2008.02.001.PubMedCrossRef

41.

Scher MS, Richardson GA, Coble PA, Day NL, Stoffer DS. The effects of prenatal alcohol and marijuana exposure: disturbances in neonatal sleep cycling and arousal. Pediatr Res. 1988;24(1):101–5 https://doi.org/10.1203/00006450-198807000-00023.PubMedCrossRef

42.

Volgin DV, Kubin L. Reduced sleep and impaired sleep initiation in adult male rats exposed to alcohol during early postnatal period. Behav Brain Res. 2012;234(1):38–42 Available from: /pmc/articles/PMC3412616/?report=abstract.PubMedPubMedCentralCrossRef

43.

Sylvester L, Kapron CM, Smith C. In utero ethanol exposure decreases rapid eye movement sleep in female Sprague ± Dawley rat offspring. 2000;289:4–7 Available from: https://pubmed.ncbi.nlm.nih.gov/10899397/.

44.

Petrelli B, Weinberg J, Hicks GG. Effects of prenatal alcohol exposure (PAE): Insights into FASD using mouse models of PAE. Biochem Cell Biol. 2018;96:131–47 Available from: https://pubmed.ncbi.nlm.nih.gov/29370535/.PubMedPubMedCentralCrossRef

45.

Ciafrè S, Ferraguti G, Greco A, Polimeni A, Ralli M, Ceci FM, et al. Alcohol as an early life stressor: epigenetics, metabolic, neuroendocrine and neurobehavioral implications. Neurosci Biobehav Rev. 2020;118:654–68 Available from: https://pubmed.ncbi.nlm.nih.gov/32976915/.PubMedCrossRef

46.

McQuire C, Daniel R, Hurt L, Kemp A, Paranjothy S. The causal web of foetal alcohol spectrum disorders: a review and causal diagram. Eur Child Adolesc Psychiatry. 2020;29:575–94 Available from: https://pubmed.ncbi.nlm.nih.gov/30648224/.PubMedCrossRef

47.

Spadoni AD, McGee CL, Fryer SL, Riley EP. Neuroimaging and fetal alcohol spectrum disorders. Neurosci Biobehav Rev. 2007;31:239–45 Available from: https://pubmed.ncbi.nlm.nih.gov/17097730/.PubMedCrossRef

48.

Nuñez SC, Roussotte F, Sowell ER. Focus on: Structural and functional brain abnormalities in fetal alcohol spectrum disorders. Alcohol Res Health. 2011;34(1):121–32 Available from: /pmc/articles/PMC3860550/?report=abstract.PubMedPubMedCentral

49.

Donald KA, Eastman E, Howells FM, Adnams C, Riley EP, Woods RP, et al. Neuroimaging effects of prenatal alcohol exposure on the developing human brain: a magnetic resonance imaging review. Acta Neuropsychiatr. 2015;27(5):251–69 https://doi.org/10.1017/neu.2015.12.PubMedCrossRef

50.

Dyląg K, Sikora-Sporek A, Bańdo B, Boroń-Zyss J, Drożdż D, Dumnicka P, et al. Magnetic resonance imaging (MRI) findings among children with fetal alcohol syndrome (FAS), partial fetal alcohol syndrome (pFAS) and alcohol related neurodevelopmental disorders (ARND). Przegl Lek. 2016;73(9):605–9 Available from: https://pubmed.ncbi.nlm.nih.gov/29688653/.

51.

Olateju OI, Ihunwo AO, Manger PR. Changes to the somatosensory barrel cortex in C57BL/6J mice at early adulthood (56 days post-natal) following prenatal alcohol exposure. J Chem Neuroanat. 2019;96:49–56 https://doi.org/10.1016/j.jchemneu.2018.12.006.PubMedCrossRef

52.

Earnest DJ, Chen WJ, West JR. Developmental alcohol and circadian clock function. Alcohol Res Health. 2001;25(2):136–40 Available from: http://www.ncbi.nlm.nih.gov/pubmed/11584552.PubMedPubMedCentral

53.

Chen CP, Kuhn P, Advis JP, Sarkar DK. Prenatal ethanol exposure alters the expression of period genes governing the circadian function of beta-endorphin neurons in the hypothalamus. J Neurochem. 2006;97(4):1026–33 https://doi.org/10.1111/j.1471-4159.2006.03839.x.PubMedCrossRef

54.

Goodlett CR, Horn KH. Mechanisms of alcohol-induced damage to the developing nervous system. Alcohol Res Health. 2001;25(3):175–84 Available from: https://pubmed.ncbi.nlm.nih.gov/11810955/.PubMedPubMedCentral

55.

Astley SJ, Clarren SK. Measuring the facial phenotype of individuals with prenatal alcohol exposure: correlations with brain dysfunction. Alcohol Alcohol. 2001;36(2):147–59 Available from: http://www.ncbi.nlm.nih.gov/pubmed/11259212.PubMedCrossRef

56.

Del Campo M, Jones KL. A review of the physical features of the fetal alcohol spectrum disorders. Eur J Med Genet. 2017;60(1):55–64 https://doi.org/10.1016/j.ejmg.2016.10.004.PubMedCrossRef

57.

Karimzadeh P. Psycho-cognitive behavioral problems in sleep disordered children. Neural Regen Res. 2012;7(8):635–9 Available from: https://pubmed.ncbi.nlm.nih.gov/25745456/.PubMedPubMedCentral

58.

CLM P, Alto ME, Hart AR, Freeze SM, Cole LL. “I’m Doing My Part, I Just Need Help From the Community”: intervention implications of foster and adoptive parents’ experiences raising children and young adults with FASD. J Fam Nurs. 2019;25(2):314–47 Available from: https://pubmed.ncbi.nlm.nih.gov/31079560/.CrossRef

Title: Sleep problems among children with Fetal Alcohol Spectrum Disorders (FASD)- an explorative study
Authors: Katarzyna Anna Dylag
Bożena Bando
Zbigniew Baran
Paulina Dumnicka
Katarzyna Kowalska
Paulina Kulaga
Katarzyna Przybyszewska
Jakub Radlinski
Sylvia Roozen
Leopold Curfs
Publication date: 01-12-2021
Publisher: BioMed Central
Keywords: Polysomnography
Fetal Alcohol Spectrum Disorder
Published in: Italian Journal of Pediatrics / Issue 1/2021
Electronic ISSN: 1824-7288
DOI: https://doi.org/10.1186/s13052-021-01056-x

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Sleep problems among children with Fetal Alcohol Spectrum Disorders (FASD)- an explorative study

Abstract

Background

Methods

Results

Conclusion

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Abstract

Background

Methods

Results

Conclusion

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